Liu X, Wu H, Byrne M, Krane S, Jaenisch R
The Whitehead Institute for Biomedical Research, Massachusetts Institute of Technology, Cambridge 02142, USA.
Proc Natl Acad Sci U S A. 1997 Mar 4;94(5):1852-6. doi: 10.1073/pnas.94.5.1852.
Type III collagen is a fibrillar forming collagen comprising three alpha1(III) chains and is expressed in early embryos and throughout embryogenesis. In the adult, type III collagen is a major component of the extracellular matrix in a variety of internal organs and skin. Mutations in the COL3A1 gene have been implicated as a cause of type IV Ehlers-Danlos syndrome, a disease leading to aortic rupture in early adult life. To directly study the role of Col3a1 in development and disease, we have inactivated the Col3a1 gene in embryonic stem cells by homologous recombination. The mutated allele was transmitted through the mouse germ line and homozygous mutant animals were derived from heterozygous intercrosses. About 10% of the homozygous mutant animals survived to adulthood but have a much shorter life span compared with wild-type mice. The major cause of death of mutant mice was rupture of the major blood vessels, similar to patients with type IV Ehlers-Danlos syndrome. Ultrastructural analysis of tissues from mutant mice revealed that type III collagen is essential for normal collagen I fibrillogenesis in the cardiovascular system and other organs.
III型胶原蛋白是一种形成纤维的胶原蛋白,由三条α1(III)链组成,在早期胚胎及整个胚胎发育过程中均有表达。在成体中,III型胶原蛋白是多种内脏器官和皮肤细胞外基质的主要成分。COL3A1基因突变被认为是IV型埃勒斯-当洛综合征的病因,这是一种在成年早期会导致主动脉破裂的疾病。为了直接研究Col3a1在发育和疾病中的作用,我们通过同源重组在胚胎干细胞中使Col3a1基因失活。突变等位基因通过小鼠生殖系传递,纯合突变动物由杂合子杂交产生。约10%的纯合突变动物存活至成年,但与野生型小鼠相比寿命短得多。突变小鼠的主要死因是主要血管破裂,这与IV型埃勒斯-当洛综合征患者相似。对突变小鼠组织的超微结构分析表明,III型胶原蛋白对心血管系统和其他器官中正常的I型胶原纤维形成至关重要。